Recruitment of CXCR4+ type 1 innate lymphoid cells distinguishes sarcoidosis from other skin granulomatous diseases

J Clin Invest. 2024 Sep 3;134(17):e178711. doi: 10.1172/JCI178711.

Abstract

Sarcoidosis is a multiorgan granulomatous disease that lacks diagnostic biomarkers and targeted treatments. Using blood and skin from patients with sarcoid and non-sarcoid skin granulomas, we discovered that skin granulomas from different diseases exhibit unique immune cell recruitment and molecular signatures. Sarcoid skin granulomas were specifically enriched for type 1 innate lymphoid cells (ILC1s) and B cells and exhibited molecular programs associated with formation of mature tertiary lymphoid structures (TLSs), including increased CXCL12/CXCR4 signaling. Lung sarcoidosis granulomas also displayed similar immune cell recruitment. Thus, granuloma formation was not a generic molecular response. In addition to tissue-specific effects, patients with sarcoidosis exhibited an 8-fold increase in circulating ILC1s, which correlated with treatment status. Multiple immune cell types induced CXCL12/CXCR4 signaling in sarcoidosis, including Th1 T cells, macrophages, and ILCs. Mechanistically, CXCR4 inhibition reduced sarcoidosis-activated immune cell migration, and targeting CXCR4 or total ILCs attenuated granuloma formation in a noninfectious mouse model. Taken together, our results show that ILC1s are a tissue and circulating biomarker that distinguishes sarcoidosis from other skin granulomatous diseases. Repurposing existing CXCR4 inhibitors may offer a new targeted treatment for this devastating disease.

Keywords: Cellular immune response; Dermatology; Immunology; Innate immunity.

MeSH terms

  • Animals
  • Chemokine CXCL12 / genetics
  • Chemokine CXCL12 / immunology
  • Chemokine CXCL12 / metabolism
  • Female
  • Granuloma* / immunology
  • Granuloma* / pathology
  • Humans
  • Immunity, Innate*
  • Lymphocytes / immunology
  • Lymphocytes / pathology
  • Male
  • Mice
  • Receptors, CXCR4* / genetics
  • Receptors, CXCR4* / immunology
  • Receptors, CXCR4* / metabolism
  • Sarcoidosis* / immunology
  • Sarcoidosis* / pathology
  • Signal Transduction / immunology
  • Skin / immunology
  • Skin / pathology
  • Skin Diseases / immunology
  • Skin Diseases / pathology

Substances

  • Receptors, CXCR4
  • CXCR4 protein, human
  • CXCR4 protein, mouse
  • Chemokine CXCL12
  • CXCL12 protein, human